Benign Acute Childhood Myositis
http://www.100md.com
《美国医学杂志》
Kanchi Kamakoti CHILDS Trust Hospital, Chennai, India, India
Abstract
Abstract. Objective: To describe the clinical and laboratory features of benign acute childhood myositis. Methods : 40 children of BACM were seen during October 2001 to February 2002, 22 (52%) were male with mean age of 5.3 years. Duration of illness was 3.97 days. Preceding symptoms included fever, leg pain, vomiting and inability to walk. A provisional diagnosis of viral myositis was made in 26 (66%). Guillian Barre Syndrome was the most common referral diagnosis. Results: 11 (27.5%) children had leucopenia with lymphocytic response and 16 (40%) had thrombocytopenia. CRP was negative in 32 (80%). CPK was markedly elevated (more than 1000 IU/l) in 18 (45%) and more than 500IU/l in 11 (27.5%) remaining between 200 to 500IU/l. Associated features were hepatitis (elevated SGOT & SGPT) in 28 (70%) and shock in 5 (12.5%).Serological test were indicative of dengue virus (Elisa PAN BIO) in 20 (50%) of which 8 (25%) were primary dengue and 12 (30%) were secondary dengue. The outcome of therapy mainly supportive were excellent. Conclusion: Benign acute myositis occurs often in association with viral infection. In the present study, Dengue virus was positive in 20 (50%) children. Benign acute myositis can be differentiated from more serious causes of walking difficulty by presence of calf and thigh muscle tenderness on stretching, normal power and deep tendon reflex and elevated CPK. [Indian J Pediatr 2005; 72 (5) : -400]
Keywords: Myositis; Dengue virus; Guillian Barre Syndrome
Benign acute childhood myositis (BACM) was first described in 1957 by Lundberg[1] under the name of "Myalgia Cruris Epidemica". It is characterised by sudden onset of calf pain and refusal or difficulty to walk. Viral agents have been implicated. [1, 2, 3, 4] It is followed by rapid recovery within a week. It is rarely reported in adults. We report 40 cases of BACM between October 2001 to February 2002.
Material and methods
0Inclusion criteria: (i) Children presenting with fever / myalgia. (ii) Muscle tenderness demonstrated by straight leg raising test (iii) Absence of nuchal rigidity. (iv) Absence of neurological signs such as altered sensorium or local deficit.
Results
0Forty cases of BACM were seen. 22 (55 % ) were male with mean age of 5.3 years age. All cases were seen between to October 2001 - February 2002. Duration of illness was 3.97 days. Preceding symptoms included fever, leg pain, headache.
A provisional diagnosis of viral myositis was made in 26 (65 % ). Guillian Barre syndrome was the most common referral diagnosis in 5 (12.5 % ). 18 (45 % ) children were unable to walk and in those who walked a characteristic pattern were observed, they walked on toes or walked with wide based, stiff gait. Muscle tenderness was recorded in 34 (85 % ) cases. This was well illustrated by stretching of the muscle by raising the leg. Similar observations were made by Mackary et al.[5]
Eleven children (27.5%) had leucopenia, with lymphocyte response 16 (40%) had thrombocytopenia. CRP was negative in 32 (80%). CPK was markedly elevated (more than 1000 IU/litre) in 18 (45%), and more than 500 IU/litre in 11 (27.5%) [Fig 1]. Serological tests were indicative of dengue virus (Elisa PAN BIO) primary Dengue in 8 (25%), secondary Dengue in 12 (30%).
Associated features were hepatitis in 28 (70%) (elevated SGOT and SGPT) shock in 5 (25.5%).
The outcome of the therapy was excellent, everybody recovered. Repeat CPK was normal in 10 (25%). Rest of the children were asymptomatic at review after two weeks. Therapy was mainly supportive with intravenous fluids as indicated and paracetamol.
Discussion
0After the first reports of BACM in 1957[1], Middleton et al[2] described cases in 1970 and showed elevated CPK in association with influenza B. Since then there have been 253 cases reported associated with various virus infections.
In the present series 55% of patients were boys. In the report by Mackary et al 84% of patients were boys. This may be either due to greater levels of activity in boys or a genetic predisposition.
The mean age of onset of symptoms in our study was 5.3 days. The mean duration of prodromal symptoms in the present study was 3.97 days. There was associated clinical features of hepatomegaly in 33 (82.5%) cases. Shock in 5 (12.5%). Resolution was rapid within as week. This was similar to the observation of Mackary et al and Mckinlay et al. Myalgia primarily affected the gastroneomius and hamstring muscles.
Viruses associated with myositis according to several authors include Influenza A, adenovirus , Coxsackie and para influenza. In the present series the Dengue virus was associated with 20 (50%) cases of myositis[6].
Muscle biopsies performed in myositis showed non specific degenerative changes, focal muscle fibre vacuolation and muscle necrosis. The elevation of CPK suggests muscle fibre destruction in keeping with a myositic component.
Awareness of the possibility of myositis in children presenting with acute myalgia, difficulty in walking in association with elevated CPK may lead to streamlining further tests and unnecessary therapy may be avoided.
Contributions
SR drafted the manuscript. KR collected the data. SE analysed the data. SR will act as the guarantor of the paper
References
1. Lundberg A Myalgia cruris epidermica. Acta Paediatr 1957;46 : 18-31.
2. Middleton PJ, Alexander RM. Severe myositis during recovering from influenza. Lancet 1970; 2 : 533-535.
3. Anthony J H, Procopis PG, Ouvier RA. Benign acute childhood myositis. Neurology 1979; 29 : 1068-1071.
4. Mckinlay IA, Mitchell I. Transient acute myositis in childhood. Arch Dis Child 1976, 51; 135-137.
5. Mackay MT, Kornverg AJ, Shield LK, Dennett X. Benign acute childhood myositis. Neurology 1999; 53 : 70.
6. Mark Barasz. Benign acute myositis. Am J Emerg Med 2000;18 : 35-37(Rajajee Sarala, Ezhilaras)
Abstract
Abstract. Objective: To describe the clinical and laboratory features of benign acute childhood myositis. Methods : 40 children of BACM were seen during October 2001 to February 2002, 22 (52%) were male with mean age of 5.3 years. Duration of illness was 3.97 days. Preceding symptoms included fever, leg pain, vomiting and inability to walk. A provisional diagnosis of viral myositis was made in 26 (66%). Guillian Barre Syndrome was the most common referral diagnosis. Results: 11 (27.5%) children had leucopenia with lymphocytic response and 16 (40%) had thrombocytopenia. CRP was negative in 32 (80%). CPK was markedly elevated (more than 1000 IU/l) in 18 (45%) and more than 500IU/l in 11 (27.5%) remaining between 200 to 500IU/l. Associated features were hepatitis (elevated SGOT & SGPT) in 28 (70%) and shock in 5 (12.5%).Serological test were indicative of dengue virus (Elisa PAN BIO) in 20 (50%) of which 8 (25%) were primary dengue and 12 (30%) were secondary dengue. The outcome of therapy mainly supportive were excellent. Conclusion: Benign acute myositis occurs often in association with viral infection. In the present study, Dengue virus was positive in 20 (50%) children. Benign acute myositis can be differentiated from more serious causes of walking difficulty by presence of calf and thigh muscle tenderness on stretching, normal power and deep tendon reflex and elevated CPK. [Indian J Pediatr 2005; 72 (5) : -400]
Keywords: Myositis; Dengue virus; Guillian Barre Syndrome
Benign acute childhood myositis (BACM) was first described in 1957 by Lundberg[1] under the name of "Myalgia Cruris Epidemica". It is characterised by sudden onset of calf pain and refusal or difficulty to walk. Viral agents have been implicated. [1, 2, 3, 4] It is followed by rapid recovery within a week. It is rarely reported in adults. We report 40 cases of BACM between October 2001 to February 2002.
Material and methods
0Inclusion criteria: (i) Children presenting with fever / myalgia. (ii) Muscle tenderness demonstrated by straight leg raising test (iii) Absence of nuchal rigidity. (iv) Absence of neurological signs such as altered sensorium or local deficit.
Results
0Forty cases of BACM were seen. 22 (55 % ) were male with mean age of 5.3 years age. All cases were seen between to October 2001 - February 2002. Duration of illness was 3.97 days. Preceding symptoms included fever, leg pain, headache.
A provisional diagnosis of viral myositis was made in 26 (65 % ). Guillian Barre syndrome was the most common referral diagnosis in 5 (12.5 % ). 18 (45 % ) children were unable to walk and in those who walked a characteristic pattern were observed, they walked on toes or walked with wide based, stiff gait. Muscle tenderness was recorded in 34 (85 % ) cases. This was well illustrated by stretching of the muscle by raising the leg. Similar observations were made by Mackary et al.[5]
Eleven children (27.5%) had leucopenia, with lymphocyte response 16 (40%) had thrombocytopenia. CRP was negative in 32 (80%). CPK was markedly elevated (more than 1000 IU/litre) in 18 (45%), and more than 500 IU/litre in 11 (27.5%) [Fig 1]. Serological tests were indicative of dengue virus (Elisa PAN BIO) primary Dengue in 8 (25%), secondary Dengue in 12 (30%).
Associated features were hepatitis in 28 (70%) (elevated SGOT and SGPT) shock in 5 (25.5%).
The outcome of the therapy was excellent, everybody recovered. Repeat CPK was normal in 10 (25%). Rest of the children were asymptomatic at review after two weeks. Therapy was mainly supportive with intravenous fluids as indicated and paracetamol.
Discussion
0After the first reports of BACM in 1957[1], Middleton et al[2] described cases in 1970 and showed elevated CPK in association with influenza B. Since then there have been 253 cases reported associated with various virus infections.
In the present series 55% of patients were boys. In the report by Mackary et al 84% of patients were boys. This may be either due to greater levels of activity in boys or a genetic predisposition.
The mean age of onset of symptoms in our study was 5.3 days. The mean duration of prodromal symptoms in the present study was 3.97 days. There was associated clinical features of hepatomegaly in 33 (82.5%) cases. Shock in 5 (12.5%). Resolution was rapid within as week. This was similar to the observation of Mackary et al and Mckinlay et al. Myalgia primarily affected the gastroneomius and hamstring muscles.
Viruses associated with myositis according to several authors include Influenza A, adenovirus , Coxsackie and para influenza. In the present series the Dengue virus was associated with 20 (50%) cases of myositis[6].
Muscle biopsies performed in myositis showed non specific degenerative changes, focal muscle fibre vacuolation and muscle necrosis. The elevation of CPK suggests muscle fibre destruction in keeping with a myositic component.
Awareness of the possibility of myositis in children presenting with acute myalgia, difficulty in walking in association with elevated CPK may lead to streamlining further tests and unnecessary therapy may be avoided.
Contributions
SR drafted the manuscript. KR collected the data. SE analysed the data. SR will act as the guarantor of the paper
References
1. Lundberg A Myalgia cruris epidermica. Acta Paediatr 1957;46 : 18-31.
2. Middleton PJ, Alexander RM. Severe myositis during recovering from influenza. Lancet 1970; 2 : 533-535.
3. Anthony J H, Procopis PG, Ouvier RA. Benign acute childhood myositis. Neurology 1979; 29 : 1068-1071.
4. Mckinlay IA, Mitchell I. Transient acute myositis in childhood. Arch Dis Child 1976, 51; 135-137.
5. Mackay MT, Kornverg AJ, Shield LK, Dennett X. Benign acute childhood myositis. Neurology 1999; 53 : 70.
6. Mark Barasz. Benign acute myositis. Am J Emerg Med 2000;18 : 35-37(Rajajee Sarala, Ezhilaras)